We report a 14-month-old toddler admitted to the Pediatric Oncology Department after surgical resection of supratentorial anaplastic ependymoma. The child was treated with International Society of Pediatric Oncology Ependymoma II 2015 chemotherapy protocol (vincristine, carboplatin, cisplatin, cyclophosphamide and methotrexate). At the end of the first cycle the child presented with symptoms such as unsteadiness and ataxic gait along with decreased motor and sensory action potentials of the limbs. As the father of the child was diagnosed with Charcot-Marie-Tooth 1A disease, a genetic analysis of the PMP22 gene was performed confirming the diagnosis of Charcot- Marie-Tooth 1A in the child, too. This case gently reminds the possibility of vincristine-induced neurotoxicity and underscores the significance of an appropriate neurological assessment before vincristine initiation.

Keywords: Charcot-Marie-Tooth, ependymoma, neurooncology, neurotoxicity, vincristine